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Dentin Dysplasia Type I: Report of Two Cases 1Zahra Elmi R, 2Somaye Azimi, 3Soudeh Jafari, 4Amir Farzanegan

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Dentin Dysplasia Type I: Report of Two Cases 1Zahra Elmi R, 2Somaye Azimi, 3Soudeh Jafari, 4Amir Farzanegan IJEDS Zahra Elmi R et al 10.5005/jp-journals-10029-1153 CASE REPORT Dentin Dysplasia Type I: Report of Two Cases 1Zahra Elmi R, 2Somaye Azimi, 3Soudeh Jafari, 4Amir Farzanegan ABSTRACT dentitions. The DD type I is more common. The DDI Objectives: In this article, we report two cases of type I dentin also called “rootless teeth” is characterized by the dysplasia (DD): the first case is subtype “d” DD and second presence of teeth with normal appearing crowns, but case is severe form of DD type I, which presents all of four no or only rudimentary root development, incomplete subtypes and describes characteristics and the management or total obliteration of the pulp chamber, and periapical of this type of teeth anomalies. radiolucent areas or cysts. In this anomaly, teeth may Materials and methods: Dentin dysplasia is a rare abnormality 4,5 of dentin that involves both primary and permanent dentition. exhibit extreme mobility and exfoliate prematurely. The normal shape of crown, short roots or its total absence, pulp The DDII, called coronal DD, is characterized by partial chamber obliteration, crescent-shaped pulp, pulp stone, and pulpal obliteration with the amorphous dentin often form- periapical radiolucencies are the characteristics of DD type I. ing pulp stones, thistle-tube-shaped coronal pulp chambers, Conclusion: There are many problems in the management of thread-like root canals, and the absence of periapical radio- a patient with DD. Depending on root development and pulp canal situation, endodontic treatment and tooth extraction are lucencies. Primary teeth with complete pulp obliteration applied in teeth with pulp necrosis and periapical abscess. The and brown or amber bluish coloration are observed similar prognosis of DD largely depends upon the age at which the to that seen in hereditary opalescent dentin. The permanent diagnosis is made, and the quality of the treatment provided. teeth have a normal appearance or a slight amber coloration; Keywords: Dentin dysplasia, Pulp obliteration, Rootless teeth. the roots are normal in size and shape.4,6 How to cite this article: Elmi R Z, Azimi S, Jafari S, This report describes two cases of DDI, clinical and Farzanegan A. Dentin Dysplasia Type I: Report of Two Cases. Int J Experiment Dent Sci 2017;6(1):44-47. radiographic view of this abnormality in detail. Source of support: Nil Conflict of interest: None CASE REPORTS Case 1 INTRODUCTION A 24-year-old male was referred to the Oral Medicine Dentin dysplasia (DD), the autosomal dominant heredi- Department of Shahid Beheshti University, Faculty of tary disturbance in dentin formation, is a rare anomaly Dentistry, Tehran, for routine oral examination. The with unknown etiology, which occurs approximately 1 patient’s medical history showed no remarkable problem in every 100,000 in the world.1,2 but in habitual history, he had bruxism and mouth breath- The first case reported with this condition was in ing. The profile was concave, the face had long appear- 1920 termed as “rootless teeth”, and in 1939, Rushton ance, and in the extraoral examination, the lower face termed this condition as DD.3 Dentinal dysplasia is had asymmetry. Intraoral examination showed severe classified into radicular dentin dysplasia (DDI) and maxillary constriction, class III occlusion, and crowding coronal dysplasia (DDII). Both types include generalized/ in anterior teeth of maxilla and mandible. The crowns had multiple involvements of the primary and permanent normal appearance (Fig. 1). The patient’s oral hygiene was moderate. There was gingivitis, amalgam and composite fillings, and prosthetic restoration. 1-3 4 Assistant Professor, Postgraduate Student Panoramic view showed normal coronal enamel 1Department of Oral and Maxillofacial Medicine, School of and dentin, normal pulp chamber size, but short and Dentistry, Guilan University of Medical Sciences, Rasht, Islamic malformed roots prominently in molars and premolars. Republic of Iran Periapical radiolucency of maxillary second premolar and 2,3Department of Oral Medicine, Shahid Beheshti University of Medical Sciences, Tehran, Islamic Republic of Iran left first molar with superficial filling was seen (Fig. 2). Another periapical radiolucency like left lateral incisor, 4Department of Periodontics, Yazd University of Medical Sciences, School of Dentistry, Yazd, Islamic Republic of Iran right second premolar, and first molars had the same condition, which seemed related to the bad fillings or Corresponding Author: Amir Farzanegan, Postgraduate Student Department of Periodontics, Yazd University of Medical Sciences incomplete root canal therapy. However, the clinical and School of Dentistry, Yazd, Islamic Republic of Iran, e-mail: radiographic pattern of dentition defined DD type I as an [email protected] appropriate diagnosis. 44 IJEDS Dentin Dysplasia Type I: Report of Two Cases A B Figs 1A and B: (A) Patient’s profile; and (B) severe crowding and maxillary constriction Case 2 An 18-year-old man attended the Oral Medicine Depart- ment of Shahid Beheshti Dental School, Tehran, with a complaint of teeth mobility and premature exfoliation of the teeth. Medical history was unremarkable. Extraoral examination showed no abnormality, competent lips, and normal profile. Intraoral examination revealed the absence of most upper teeth except two molars and one premolar, and some remaining roots. Most of the lower teeth were present (Fig. 3). On examination, molars and lower incisors show grade II mobility and premolars have grade III mobility Fig. 2: Panoramic view of multiple periapical radiolucency and except the lower right second premolar. The patient’s shortening of the roots, especially in the posterior teeth oral hygiene was poor and all posterior remaining teeth had caries or filling. Plaque and calculus were The patient had no complaint about profile pattern of present. occlusion. Treatment plan consisted of determination of Radiographic examinations revealed teeth with periodontal prognosis for lower first molars, endodontic very short, blunt, and malformed roots. Rudimentary treatment for teeth with periapical radiolucency, root formation in upper and lower anterior teeth and instructing oral hygiene, scaling, and root planing. molars was seen and all premolars except the lower A B Figs 3A and B: (A) Normal profile; and (B) most of the teeth lost in the upper jaw International Journal of Experimental Dental Science, January-June 2017;6(1):44-47 45 Zahra Elmi R et al permanent teeth and total pulp chamber obliteration in primary teeth.8 OCarroll categorized DDI as having four radiographic variations in permanent teeth. The variations are based on the amount of obliteration of the pulp chambers, root development, and periapical radiolucent areas. In the first category, DDIa, there is the complete obliteration of the pulp, no root development, and many periapical radiolucent areas; DDIb and DDIc have less pulpal obliteration and feature crescent-shaped radiolucent areas in the chamber, more root development, and less frequent Fig. 4: Panoramic view shows the loss of a large number of teeth, periapical radiolucencies. In DDId, the pulp chambers are too short roots, and absence of root. Obliterated pulp and crescent- distinct, with pulp stones in the coronal third of the root shaped pulp chamber canal (with corresponding bulging of the root); the root has significant development, and there are few, if any, right second premolar showed no root formation or periapical radiolucencies.7,9 malformed roots (Fig. 4). By this categorization, case 1 is placed in subtype DDId, There was an impacted malformed canine. Lower and case 2 contains all subtypes of DDI. The radiographic primary canine in the left side still remained. All teeth characteristics of the permanent dentition can be used to showed pulp chamber obliteration. Crescent-shaped pulp differentiate DDI from DDII.9 Teeth with radiographic chamber in the lower third molars was seen, which is or histologic features of DD occur in a number of characteristic of DDI. disorders, such as calcinosis, osteogenesis imperfecta, Furthermore, radiographic findings showed periapical Ehlers–Danlos syndrome, Goldblatt syndrome, Schimke radiolucencies and many remaining roots in the jaws. immuno-osseous dysplasia, and Brachio-skeletal-genital There was no family history of similar dental syndrome.8 abnormality and also finding did not support the Azma et al10 report a case of DD type II associated presence of systemic disorders. with hypoplastic amelogenesis imperfecta in an 18-year- Treatment strategies consisted of patient’s information old girl, in Rasht, Guilan, without any syndromic signs. In to improve oral health, extraction of all the mobile and explaining this coincidence, some studies have proposed periodontally hopeless teeth, and remaining roots. Root that DD and dentinogenesis imperfecta represent allelic canal therapy due to pulp chamber obliteration was not mutations of a common gene. feasible. Toomarian et al11 present a case of DD type I in a In order for mastication and esthetics, a provisional 12-year-old Iranian boy. Radiographic examination re- treatment was considered. Implant-supported pros- vealed pulpless teeth with no root formation in most thesis considering bone graft was advised as a final teeth. There was a radicular cyst in association with a treatment. permanent molar. Shokri et al12 in 2014 performed a cross-sectional DISCUSSION study on 1649 people in Iran, Hamadan, to evaluate the The DD is a genetic defect of dentin formation, although dental anomalies among 7- to 35-year-old patients using its etiology is still considered a mystery.1 Wesley et al7 panoramic radiographs. The frequency of DD was 1 in proposed that the condition is caused by an abnormal 1,649 people with a prevalence of 0.06%. interaction of odontoblasts with ameloblasts, leading to The management of patients with DD has presented abnormal differentiation and/or function of the ecto- dentists with many problems. Extraction has been mesenchymal-derived odontoblasts. The DD is usually suggested as an alternative treatment for teeth with pulp an autosomal dominant condition, and there is a 50% necrosis and periapical abscess.
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